Figure 1
Diffusion-weighted and Apparent Diffusion Coefficient Sequences of Magnetic Resonance Images of Brain. (A,B) Case 1: acute infarct in left basal ganglia. (C,D) Case 2: acute infarct in right basal ganglia, right corona radiata, and subcortical white matter. (E,F) Case 3: acute infarct in left basal ganglia, left corona radiata and subcortical white matter. (G,H) Case 4: acute infarct in the right basal ganglia and adjacent white matter. (I,J) Case 5: acute infarct in the left basal ganglia and adjacent white matter. (K,L) Case 6: acute infarct in the left corona radiata. (M,N) Case 7: left thalamic hemorrhage. Non-contrast computed tomography of head. (O) Case 8: chronic lacunar infarcts in the right basal ganglia and right parietal region. Axial T2 fluid-attenuated inversion recovery image. (P–R) Axial T2-weighted sequences of magnetic resonance images of brain. (P) Case 9: chronic infarcts in left posterior cerebral artery region, left thalamus and lacunar infarcts in bilateral basal ganglia). (Q) Case 10: chronic infarcts in left frontal subcortical region. (R) Case 11: chronic infarct of left middle cerebral artery region.
Figure 2
Lesions Causing Post-stroke Lingual Dystonia. This figure (top row) illustrates the imaging lesion localization in patients with post-stroke lingual dystonia described in the literature. Case 1 depicts acute post-stroke lingual dystonia, cases 2 and 3 are patients with chronic post-stroke oromandibular (lingual) dystonia, and case 4 is a patient with chronic post-stroke cranial lingual dystonia. The bottom two rows illustrate the imaging lesion localization in our patients with post-stroke abnormal lingual movements. Cases 1–7 are acute post-stroke lingual dystonia patients whereas cases 8–11 are chronic post-stroke lingual dystonia patients. Red lesions represent hemorrhagic stroke and blue lesions represent acute ischemic stroke whereas brown represents chronic ischemic stroke.
Video 1.
Post-stroke Lingual Dystonia. Lingual dystonia cases 1–11.
Table 1
Clinical and Neuroimaging Details of Post-stroke Lingual Dystonia Patients
| Case Number | Age and Sex | Handedness | Risk Factors for Stroke | Days of Onset | Lingual Dystonia | Tongue Tremor | Neuroimaging (All MRI Except Case 7) | Outcome |
|---|---|---|---|---|---|---|---|---|
| 1. | 70 years, Female | Right | None | 1 day | Severe on rest and mild on protrusion | On protrusion | Acute infarct in the left basal ganglia region with bilateral chronic infarcts | Moderate |
| 2. | 54 years, Male | Right | HTN, chronic smoker and alcohol intake | 7 days | Severe on rest | Absent | Acute infarcts involving right basal ganglia, right corona radiata and subcortical white matter changes in fronto-parietal and periventricular regions | Mild |
| 3. | 40 years, Male | Right | HTN | 5 days | Severe on rest and mild on protrusion | Absent | Acute infarct in left basal ganglia region, left corona radiata and adjacent subcortical white matter with bilateral chronic lacunar infarcts | Excellent |
| 4. | 59 years, Male | Right | HTN, DM | 7 days | Only on rest | Absent | Acute infarct involving the right basal ganglia and periventricular white matter changes | Moderate |
| 5. | 41 years, Male | Right | DM | 2 days | Severe on rest and mild on protrusion | On protrusion | Acute infarct in the left basal ganglia region and chronic infarcts in left frontoparietal cortical-subcortical regions | Moderate |
| 6. | 45 years, Female | Right | DM, HTN, Dilated cardiomyopathy | 4 days | Both rest and protrusion | On protrusion | Acute infarct in left corona radiata regions with bilateral chronic lacunar infarcts | Excellent |
| 7. | 55 years, Male | Right | Chronic bidi smoker | 3 days | Severe on rest and mild on protrusion | Absent | CT scan of brain revealed left thalamic hemorrhage | Patient died |
| 8. | 62 years, Female | Right | Rheumatic heart disease | 24 months | Severe on rest and mild on protrusion | Tremor on protrusion | Chronic infarcts in bilateral cerebellum, left thalamus, right basal ganglia, right posterior frontal and parietal areas, and left corona radiata regions | Moderate |
| 9. | 61 years, Male | Right | Alcohol | 5 months | Both on rest and protrusion | Tremor on rest and protrusion | Chronic infarcts in bilateral basal ganglia and predominantly right temporo-parietal regions with white matter hyperintensities in bilateral periventricular areas | Mild |
| 10. | 59 years, Female | Right | DM | 18 months | Only on rest | Tremor on rest and protrusion | Chronic infarcts in left fronto-parietal, bilateral pons, basal ganglia and right frontal subcortical regions | Mild |
| 11. | 65 years, Female | Right | HTN,Cardiac apical aneurysm | 4 months | Severe on rest and mild on protrusion | Tremor on protrusion | Chronic infarct in left middle cerebral artery territory with haemorrhagic transformation | Mild |
Table 2
Neurological Examination in Post-stroke Lingual Dystonia Patients
| Case Number | Age and Sex | Neurological Examination Findings |
|---|---|---|
| 1. | 70 years, Female | Left hemiparesis (MRC power grade 4-/5) |
| 2. | 54 years, Male | Normal motor power, left upper limb dystonic tremors, and right laterocollis |
| 3. | 40 years, Male | Right sided brachiofacial weakness (MRC power grade 4/5), right upper limb dystonia, laterocollis |
| 4. | 59 years, Male | Left sided brachiofacial weakness (MRC power grade 4/5) |
| 5. | 41 years, Male | Right upper limb weakness (MRC power grade 4+/5) |
| 6. | 45 years, Female | Left hemiparesis and dysphagia (MRC power grade 4-/5), left upper limb dystonic tremors |
| 7. | 55 years, Male | Right sided hemiplegia (MRC power grade 2/5) |
| 8. | 62 years, Female | Normal |
| 9. | 61 years, Male | Alexia, residual right hemiparesis (MRC power grade 4+/5), brisk reflexes, right upper limb dystonia and dystonic tremors, short shuffling gait and postural instability |
| 10. | 59 years, Female | Right sided brachiofacial weakness (MRC power grade 4/5) |
| 11. | 65 years, Female | Right sided hemiparesis (MRC power grade 4-/5) including mild weakness of face and tongue |
Table 3
A Comparison of Demographic and Clinical Details of Acute and Chronic Post-stroke Lingual Dystonia Patients
| Acute Presentation | Chronic Presentation | |
|---|---|---|
| Number of patients | 7 | 4 |
| Male: Female | 5:2 | 1:3 |
| Age | 40–70 (Mean ± SD: 52 ±4.08) years | 59–65 (Mean ± SD: 61.75 ±1.25) years |
| Duration of illness | 1–7 (Mean ± SD: 4.14 ± .88) days | 4–24 (Mean ± SD: 12.75±4.92) months |
| Tongue dystonia at rest | 7/7 (100%) | 4/4 (100%) |
| Tongue dystonia at protrusion | 5/7 (71.42%) | 3/4 (75%) |
| Tongue tremor | 3/7 (42.85%) | 4/4 (100%) |
| Type of stroke | ||
| Ischemic | 6 | 4 |
| Hemorrhagic | 1 | 0 |
| Neuroimaging findings | Acute lesion: 5 had left sided and 2 had right sided involvement | 3 had bilateral and 1 had left side involvement |
| Outcome | ||
| Excellent | 2 | 0 |
| Moderate | 3 | 1 |
| Mild | 1 | 3 |
| Died | 1 | 0 |
Table 4
Neuroimaging Findings in Acute and Chronic Patients with Post-stroke Lingual Dystonia
| Case | Imaging | Haemorrhage/Infarct | Cortical | Subcortical White Matter Including Corona Radiata | Basal Ganglia | Thalamus | Subthalamus | Brainstem | Cerebellum | |||||||
|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
| L | R | L | R | L | R | L | R | L | R | L | R | L | R | |||
| 1 | MRI | Acute and chronic infarct | Ch | Ch | Ch | Ch | AcCh | Ch | Ch | Ch | – | – | – | |||
| 2 | MRI | Acute and chronic infarct | – | Ch | Ac, Ch | Ac | – | – | – | – | ||||||
| 3 | MRI | Acute and chronic infarct | – | Ac, Ch | Ch | Ac | Ch | – | – | – | – | |||||
| 4 | MRI | Acute and chronic infarct | – | Ch | Ac, Ch | Ac | – | – | – | – | ||||||
| 5 | MRI | Acute and chronic infarct | Ch | – | Ch | Ac | – | – | – | – | ||||||
| 6 | MRI | Acute and chronic infarct | Ch | Ch | Ac, Ch | Ch | – | – | – | – | Ch | |||||
| 7 | NCCT | Haemorrhage | – | – | – | Ac | – | – | – | |||||||
| 8 | MRI | Chronic infarct | Ch | Ch | Ch | Ch | Ch | – | – | Ch | Ch | |||||
| 9 | MRI | Chronic infarct | Ch | Ch | Ch | Ch | Ch | Ch | Ch | – | – | – | ||||
| 10 | MRI | Chronic infarct | Ch | Ch | Ch | Ch | Ch | – | – | Ch | Ch | – | ||||
| 11 | MRI | Chronic infarct | Ch | Ch | – | – | – | – | – | |||||||
Table 5
Cases of Post-stroke Lingual Movement Disorders Reported in the Literature
| Author/Year | Serial Number of Cases | Number of Cases | Age/Sex | Type of Movement | Lesion Site/Type | Acute/Chronic |
|---|---|---|---|---|---|---|
| Dewey 1989 | 1 | One | 63 years, male | Oro-mandibular lingual dyskinesia, | Left Thalamic haemorrhage | Acute |
| O Combarros 1990 | 2 | One | 68 years, female | Oral dyskinesia (lingual dyskinesia) | Bilateral thalamo-capsular infarction | Acute |
| Jagota 2010 | 3 | One | 26 years, female | Lingual myoclonus | Bilateral cortical frontotemporal and insular region ischemic infarct | Acute |
| Salazar 2012 | 4 | One | 35 years, male | Palatal and lingual tremor | Left frontoparietal region (left Middle cerebral artery ischemic infarct) | Acute |
| Pandey 2015 | 5 | One | 42 years, male | Tongue tremor | Left frontoparietal infarct | Acute |
| Saito 2016 | 6–10 | Five | 75–84 years 4 females, one male | Rippling tongue movements | 4 with left primary motor cortex ischemic infarct and 1 with right primary motor cortex ischemic infarct | Acute |
| Rao 2018 | 11–12 | Two | 75 years, one male and one female | Lingual myoclonus | Left frontal opercular ischemic infarct, Right high frontal region ischemic infarct | Acute |
| Reilly 1992 | 13 | One | 50 years, female | Lingual chorea | Bilateral paramedian thalamic infarct (left>right) | Chronic |
| Fabiano 2000 | 14 | One | 64 years, female | Rhythmic tongue movements | Ischemic infarct in centrum semiovales and cerebral peduncles | Chronic |
| Lee 2005 | 15 | One | 82 years, female | Lingual dyskinesia | Right ventrolateral portion of mid and superior Pontine infarct | Chronic |
| Duffey 2007 | 16 | One | 64 years, male | Palatal tremor, lingual and jaw dyskinesia | Ventral pons and bilateral cerebellar infarcts | Chronic |
| Kim 2009 | 17 | One | 70 years, male | Lingual dystonia | Right thalamic infarction | Acute |
| Alarcon 2004 | 18 | One | 36 years, male | Cranial lingual dystonia | Right frontal haematoma | Chronic |
| Akin 2014 | 19 | One | 64 years, female | Oro-mandibular dystonia (lingual dystonia) | Left Anterior Inferior Cerebellar infarct | Chronic |
| Brissaud 2016 | 20 | One | 17 months old, female | Oro-mandibular dystonia (lingual dystonia) | Right basal ganglia infarct | Chronic |